Assessment of Burden on Family Caregivers of Children With Sickle Cell Anemia in Al Madinah Al Munawwarah, Saudi Arabia

Background Sickle cell anemia (SCA) results in various complications, necessitating continuous daily care and placing burdens on caregivers. Objectives This study aims to assess the burden on family caregivers of children with SCA and its associated factors. Materials and methods This analytical cross-sectional study was conducted in Madinah City, Saudi Arabia. We included family caregivers of children with SCA who were registered and treated at the Maternity and Child Hospital in King Salman Medical City. Data were collected from all registered files of children who received treatment for SCA. Data from participants was obtained using the validated Arabic version of the Zarit Burden Interview (ZBI). Descriptive statistics, chi-square tests, independent sample t-tests, and multivariate regression analysis were used in the statistical analysis. Results Overall, 124 caregivers participated out of 166 (response rate: 74.7%), among which 83 (66.9%) were fathers, 72 (58.1%) were aged ≥40 years, 96 (77.4%) held Saudi nationality, and 62 (50%) had a monthly income of <5000 SAR. The average daily caregiving hours were 5±4 hours, and 30 (24.2%) of children were diagnosed with associated physical or psychological diseases. The Zarit Burden Interview score indicated that 45 (36.3%) of caregivers reported no burden, whereas 51 (41.1%), 22 (17.7%), and 6 (4.8%) reported mild, moderate, and severe burden, respectively. Factors contributing to the burden included being a mother, low financial resources, non-Saudi nationality, children diagnosed with associated physical or psychological diseases, and caregiving hours. Conclusions The burden on SCA caregivers was higher for caregivers who were mothers, non-Saudis, those with lower income, and children with physical or psychological diseases, as well as more caregiving hours. Enhancing the overall well-being of families affected by the SCA burden involves creating targeted interventions and comprehensive support programs.


Introduction
Sickle cell anemia (SCA) is an inherited chronic hemoglobinopathy that results in various complications, necessitating continuous daily care [1].Approximately 300,000 children are born with SCA worldwide [2].Regional variations in prevalence exist in Saudi Arabia [3].Caring for children with SCA introduces difficulties for caregivers [4], with many exhibiting psychological distress [5,6].A previous study revealed that 54% of caregivers were worried about worsening symptoms [7].Various caregiving responsibilities for children with SCA, such as administering medications, providing assistance during disease complications and crises, transporting the children to clinics and hospitals for treatment and visits, as well as managing communication with healthcare providers and schools, contribute to caregiver burden [5,8,9].

Data collection
The data collection method involved contacting eligible participants via phone calls to obtain permission to participate in the study and ensure they understood the purpose of the study.Upon confirmation of their participation, a clear message was sent through WhatsApp, along with a Google Forms link containing the questionnaire and consent form, requesting that they complete it.Participants who did not respond initially were sent follow-up phone calls and messages as reminders to ensure participation.

Study tool
A self-administered questionnaire comprising three parts was used as the study tool.The first and second parts included the sociodemographic data of caregivers and children, respectively, whereas the third part included the validated Arabic version of the Zarit Burden Interview (ZBI) for the assessment of caregiver burden.The ZBI questionnaire was distributed in Arabic and constituted 22 questions, each rated on a 5point Likert-type scale (0 [never] to 4 [nearly always]; total score range: 0-88).The level of burden was classified according to the total score as follows: 0-20, little or no burden; 21-40, mild to moderate burden; 41-60, moderate to severe burden; and 61-88, severe burden [10].
The ZBI was initially developed in 1980 to evaluate the burden experienced by the caregivers of patients with Alzheimer's disease.The ZBI has since become a widely used tool for evaluating caregiver burden and has been adapted into multiple languages.It has been extensively applied in clinical studies with published reports on caregivers of individuals with various physical and mental conditions, including SCA [10,11].The psychometric properties of the ZBI have been extensively evaluated in caregivers, and the validity parameters for the Arabic version of the ZBI (Cronbach's alpha = 0.8) and Arabic translations of the ZBI have good internal consistency and reliability [12].In this study, caregiver burden was defined as a ZBI score of >41.

Statistical analysis
All statistical analyses were performed using SPSS Statistics (version 29).The distribution of participants' responses to questionnaire items was reported using descriptive statistics.Continuous data are presented as means and standard deviations, whereas categorical data are expressed as frequencies and percentages.The chi-squared and independent sample t-tests were used for inferential statistics.A multivariate logistic regression analysis was conducted.A 95% confidence interval (CI) was calculated, and statistical significance was set at a p-value of <0.05.The level of burden was assessed using the ZBI (the dependent variable) and categorized into two groups: no burden (scores of 0 to 40) and burden (scores of 41 to 88).
Regarding factors associated with caregiver burden for children with SCA, mothers were more likely to experience burden than fathers (odds ratio [OR]: 3.8, 95% CI: 1.58-9.10).Non-Saudi caregivers exhibited a higher likelihood of burden than Saudi caregivers (OR: 3.0, 95% CI: 1.20-7.56).Unemployed caregivers were more likely to experience burden than those employed in the government sector (OR: 5.1, 95% CI: 1.40-18.69).Additionally, caregivers with an income of <5000 SAR had a higher likelihood of burden than those with an income of >10000 SAR (OR: 10.3, 95% CI: 2.27-46.96)(Table 4).Mean caregiving hours per day were higher among caregivers who reported burden (8.7±4.6)compared with those among caregivers who did not (3.6±4.1;p<0.001) (Table 5).Caregivers of children diagnosed with physical or psychological diseases had a higher likelihood of experiencing burden than their counterparts (OR: 4.0, 95% CI: 1.62-9.99)(Table 6).

Discussion
Our study sheds light on the burden faced by family caregivers of children with SCA, identifying key demographic and socioeconomic factors contributing to this burden.The results show that the average number of family members, the percentage of caregivers being unemployed, and those with a low income of <5000 SAR were 6±2.5, 50.8%, and 50%, respectively.Furthermore, mothers had a higher likelihood of experiencing a burden than fathers.
Further insights from the study also examined sex distribution, age groups, and the presence of coexisting physical or psychological diseases in children with SCA and found a higher prevalence of SCA in boys (54.0%) and a higher proportion of affected children aged ≤ 9 years (54.0%).Additionally, our results reveal that caregivers of children diagnosed with physical or psychological diseases had a higher likelihood of experiencing burden, highlighting additional challenges faced by caregivers in managing younger children with coexisting health conditions.

Madani et al. reported similar characteristics among caregivers of children with SCA in western Saudi
Arabia, showing that larger families (average of 5±2.0 members) with limited financial resources (64.5%), housewife mothers (79.4%), and those lacking social assistance or health insurance (80%) experienced significant financial and emotional burdens [8].These burdens affected various aspects of their quality of life, including their social and professional lives [8].
Kuerten et al. aimed to characterize the psychosocial burden experienced by caregivers of children with SCA in Kenya and identified predictors of the psychosocial burden, including disease severity and financial hardship.Their findings align with our results, suggesting that caregivers of children with SCA experienced difficulties across multiple domains of functioning and that financial hardships were likely to be associated with psychosocial burdens [5].
Regarding caregiving hours, the study data showed that caregivers who devoted more hours to caregiving responsibilities were more likely to experience burden, suggesting that caregiving duration contributes to the overall burden experienced by caregivers.The result of this study is consistent with that of the existing literature, including a study by Madani et al. who examined the quality of life among caregivers of patients with SCA and highlighted that caregivers expressed the lowest satisfaction levels in certain areas, such as financial situation, free time activity, and life environment [8].Healthcare providers and support organizations should develop comprehensive strategies to alleviate the burden and promote overall wellbeing among caregivers by considering financial resources, caregiving hours, and the presence of coexisting health conditions.Our findings indicate that 36.3% of caregivers reported no burden, whereas 41.1%, 17.7%, and 4.8% had mild, moderate, and severe burden, respectively, based on the ZBI score.The reported caregiver burden varies among studies that used the ZBI.For example, a study conducted by Grygiel et al. in Grenada found that 53.8% of caregivers reported little to no burden, whereas 34.6% and 11.5% reported mild and moderate burden, respectively, with no reported cases of severe burden [11].By contrast, another study by Baker et al. in Jamaica reported that 26% of caregivers experienced little to no burden, whereas 61%, 12%, and 1% reported mild, moderate, and severe burden, respectively [10].
The strength of this cross-sectional study lies in selecting the Maternity and Child Hospital at King Salman Medical City in Madinah, the primary governmental hospital in the region, which provides comprehensive healthcare services and specialized care.This choice enables extensive data collection from all registered files of children receiving treatment for SCA in the Madinah region, along with a robust response rate of 74.7% from family caregivers.This approach offers valuable insights into caregiver burden and the influence of sociodemographic and clinical factors in contributing to caregiving burden.
Nevertheless, this study has some limitations, which must be considered when interpreting the findings.The cross-sectional nature of this study only provided a snapshot of caregiver burden, which may vary over time and in different disease situations.Furthermore, the single-center design restricts the applicability of our findings to a broader population.Future longitudinal studies with larger and more diverse samples should be conducted to address these limitations and further validate and generalize the findings.

Conclusions
This study highlights the importance of understanding the intricate dynamics within families affected by SCA and the sociodemographic characteristics contributing to caregiver burden.Our results indicate that caregivers of SCA children with physical or psychological diseases were more susceptible to experiencing burden, particularly regarding caregiving hours.Furthermore, caregivers with lower incomes were more likely to experience a burden.Targeted interventions and comprehensive support programs can be developed by acknowledging the presence of vulnerable populations and identifying the factors associated with caregiver burden to alleviate the burden and enhance the overall well-being of family caregivers.

First 12 -More than 12 --
part: sociodemographic data of the caregivers What is your relationship with the patient?Is there anyone from the same family (brother and sisters) who has sickle cell anemia?YES NO Is there anyone from the same family (brother and sisters) who has sickle cell anemia trait?YES NO Does anyone from the same family (brother and sister of the affected child) have a physical or psychological disease?-Post-traumatic stress disorder -Others (…………..) Do you provide health care to more than one person in the same family?YES NO How long do you care for your child measured in years?outside the hospital who helps you in providing care for the same patient?YES NO Who is the person helping you with providing care for the sickle cell anemia child? is typically spent on caregiving of the sickle cell anemia child on a daily basis, on average measured in hours?Inability to read or write -Primary 2024 Aloufi et al.Cureus 16(8): e66160.DOI 10.7759/cureus.More than 15000 SR Reason for no work -Has a relationship to your care of the patient -Has not related to your care of the patient Second Part: sociodemographic data of the child Gender Male Female Age measured in years ?How long since diagnosis with sickle cell anemia measured in years ?

TABLE 2 : Sociodemographic characteristics of children with SCA and their families: categorical variables
SCA: sickle cell anemia.

TABLE 6 : Association between caregiver burden and sociodemographic characteristics of children with SCA and their families
SCA: sickle cell anemia; OR: odds ratio; CI: confidence interval; P<0.05 significant values.